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Published in: Journal of Medical Case Reports 1/2021

Open Access 01-12-2021 | Case report

Multiple aplasia cutis congenita type V and fetus papyraceous: a case report and review of the literature

Authors: V. Thadchanamoorthy, Kavinda Dayasiri, M. Thirukumar, N. Thamilvannan, S. H. Chandraratne

Published in: Journal of Medical Case Reports | Issue 1/2021

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Abstract

Background

Aplasia cutis congenita is regarded as congenital focal absence of skin in the newborn, and occurrence of more than three similar skin defects is rare. The etiology is thought to be multifactorial, and precise etiopathogenesis is unknown.

Case presentation

A 13-day-old newborn Sri Lankan Tamil girl was referred to the dermatologic clinic with multiple skin defects at birth. There were six lesions on the body, and two of them had healed during intrauterine period, leaving scars. This was a second twin of her pregnancy. Her first twin fetus had demised before 19 weeks of pregnancy and was confirmed to be fetus papyraceous based on ultrasound-guided fetal assessment. The said child was thoroughly investigated and found to have no other congenital abnormalities. Chromosomal studies yielded normal findings. She was treated with tropical antibacterial ointment, and all lesions resolved spontaneously within 4 weeks, leaving scars. Physiotherapy was commenced to prevent contracture formation, and follow-up was arranged in collaboration with the plastic surgical team.

Conclusions

Aplasia cutis congenita is a rare condition of uncertain etiology, but consanguinity may play a role. This report described a newborn with type V cutis aplasia congenita in whom the diagnosis was confirmed based on clinical features and revision of antenatal history. The management depends on the pattern, extent, location, severity, underlying causes, and associated anomalies.
Literature
1.
go back to reference Blouin MM, Bernard J, Caron F, et al. Aplasia cutis congenita of the trunk and scalp associated with fetus papyraceus. Int J Dermatol. 2011 Jun;50(6):733–5.CrossRef Blouin MM, Bernard J, Caron F, et al. Aplasia cutis congenita of the trunk and scalp associated with fetus papyraceus. Int J Dermatol. 2011 Jun;50(6):733–5.CrossRef
2.
go back to reference Neill JK, Carter M, Warr RP. Aplasia cutis congenital. A case of scalp defect repair using two opposing bipedicled local flaps. J Plast Reconstr Aesthetic Surg. 2010;63:e242-4.CrossRef Neill JK, Carter M, Warr RP. Aplasia cutis congenital. A case of scalp defect repair using two opposing bipedicled local flaps. J Plast Reconstr Aesthetic Surg. 2010;63:e242-4.CrossRef
4.
go back to reference Duan X, Yang GE, Yu D, Yu C, et al. Aplasia cutis congenita: A case report and literature review. Exp Ther Med. 2015 Nov;10(5):1893–5.CrossRef Duan X, Yang GE, Yu D, Yu C, et al. Aplasia cutis congenita: A case report and literature review. Exp Ther Med. 2015 Nov;10(5):1893–5.CrossRef
5.
go back to reference Šimić D, Prohić A, Puizina Ivić N, et al. Aplasia cutis congenita in a newborn child associated with two fetus papyraceous. Acta Dermatovenerol Croat. 2015;23:293–7.PubMed Šimić D, Prohić A, Puizina Ivić N, et al. Aplasia cutis congenita in a newborn child associated with two fetus papyraceous. Acta Dermatovenerol Croat. 2015;23:293–7.PubMed
6.
go back to reference Brzezinski P, Pinteala T, Chiriac AE, et al. Aplasia cutis congenita of the scalp—what are the steps to be followed? Case report and review of the literature. An Bras Dermatol. 2015;90:100–3.CrossRef Brzezinski P, Pinteala T, Chiriac AE, et al. Aplasia cutis congenita of the scalp—what are the steps to be followed? Case report and review of the literature. An Bras Dermatol. 2015;90:100–3.CrossRef
7.
go back to reference Choi MSS, Choi JH, Ki SH, Jun YH. Aplasia cutis congenita associated with aplasia of the superficial temporal artery. J Craniofac Surg. 2016;27:1065–7.CrossRef Choi MSS, Choi JH, Ki SH, Jun YH. Aplasia cutis congenita associated with aplasia of the superficial temporal artery. J Craniofac Surg. 2016;27:1065–7.CrossRef
8.
go back to reference Caksen H, Kurtoglu S. Our experience with aplasia cutis congenita. J Dermatol. 2002;29:376–9.CrossRef Caksen H, Kurtoglu S. Our experience with aplasia cutis congenita. J Dermatol. 2002;29:376–9.CrossRef
9.
go back to reference Iwayama H, Hosono H, Yamamoto H, et al. Aplasia cutis congenita with skull defect in a monozygotic twin after exposure to methimazole in utero. Birth Defects Res. Part A Clin Mol Teratol. 2007;79:680–4.CrossRef Iwayama H, Hosono H, Yamamoto H, et al. Aplasia cutis congenita with skull defect in a monozygotic twin after exposure to methimazole in utero. Birth Defects Res. Part A Clin Mol Teratol. 2007;79:680–4.CrossRef
10.
go back to reference Sachs C, Tebacher-Alt M, Mark M, et al. Aplasie cutanée congénitale et antithyroïdiens de synthèse au cours de la grossesse: Série de cas et revue de la littérature. Ann Dermatol Venereol. 2016;143:423–35.CrossRef Sachs C, Tebacher-Alt M, Mark M, et al. Aplasie cutanée congénitale et antithyroïdiens de synthèse au cours de la grossesse: Série de cas et revue de la littérature. Ann Dermatol Venereol. 2016;143:423–35.CrossRef
11.
go back to reference Marcovici I. Aplasia cutis congenita presenting as vacuum-extractor-related trauma. Int J Gynecol Obstet. 2015;129:267–8.CrossRef Marcovici I. Aplasia cutis congenita presenting as vacuum-extractor-related trauma. Int J Gynecol Obstet. 2015;129:267–8.CrossRef
12.
go back to reference Garcia-Romero MT, Narvóez-Rosales V, Hojyo-Tomoka MT. Bullous aplasia cutis congenita: case report and review of the literature. Indian J Dermatol. 2011;56(3):337–8.CrossRef Garcia-Romero MT, Narvóez-Rosales V, Hojyo-Tomoka MT. Bullous aplasia cutis congenita: case report and review of the literature. Indian J Dermatol. 2011;56(3):337–8.CrossRef
13.
go back to reference Verhelle NA, Heymans O, Deleuze JP, Fabre G, Vranckx JJ, Van den hof B. Abdominal aplasia cutis congenita: case report and review of the literature. J Pediatr Surg. 2004;39(2):237–9.CrossRef Verhelle NA, Heymans O, Deleuze JP, Fabre G, Vranckx JJ, Van den hof B. Abdominal aplasia cutis congenita: case report and review of the literature. J Pediatr Surg. 2004;39(2):237–9.CrossRef
14.
go back to reference Frieden IJ. Aplasia cutis congenita: a clinical review and proposal for classification. J Am Acad Dermatol. 1986;14:646–60.CrossRef Frieden IJ. Aplasia cutis congenita: a clinical review and proposal for classification. J Am Acad Dermatol. 1986;14:646–60.CrossRef
16.
go back to reference Lemke RP, Machin G, Muttitt S, Bamforth F, Rao S, Welch R. A case of aplasia cutis congenita in dizygotic twins. J Perinatol. 1993;13(1):22–7.PubMed Lemke RP, Machin G, Muttitt S, Bamforth F, Rao S, Welch R. A case of aplasia cutis congenita in dizygotic twins. J Perinatol. 1993;13(1):22–7.PubMed
17.
21.
go back to reference Bourque S, Preloger E. Extensive aplasia cutis congenita with associated vanishing twin syndrome. J Pediatr. 2015;167(3):772.CrossRef Bourque S, Preloger E. Extensive aplasia cutis congenita with associated vanishing twin syndrome. J Pediatr. 2015;167(3):772.CrossRef
Metadata
Title
Multiple aplasia cutis congenita type V and fetus papyraceous: a case report and review of the literature
Authors
V. Thadchanamoorthy
Kavinda Dayasiri
M. Thirukumar
N. Thamilvannan
S. H. Chandraratne
Publication date
01-12-2021
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2021
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/s13256-021-02662-3

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